Literature DB >> 14755334

Lamin A/C deficiency causes defective nuclear mechanics and mechanotransduction.

Jan Lammerding1, P Christian Schulze, Tomosaburo Takahashi, Serguei Kozlov, Teresa Sullivan, Roger D Kamm, Colin L Stewart, Richard T Lee.   

Abstract

Mutations in the lamin A/C gene (LMNA) cause a variety of human diseases including Emery-Dreifuss muscular dystrophy, dilated cardiomyopathy, and Hutchinson-Gilford progeria syndrome. The tissue-specific effects of lamin mutations are unclear, in part because the function of lamin A/C is incompletely defined, but the many muscle-specific phenotypes suggest that defective lamin A/C could increase cellular mechanical sensitivity. To investigate the role of lamin A/C in mechanotransduction, we subjected lamin A/C-deficient mouse embryo fibroblasts to mechanical strain and measured nuclear mechanical properties and strain-induced signaling. We found that Lmna-/- cells have increased nuclear deformation, defective mechanotransduction, and impaired viability under mechanical strain. NF-kappaB-regulated transcription in response to mechanical or cytokine stimulation was attenuated in Lmna-/- cells despite increased transcription factor binding. Lamin A/C deficiency is thus associated with both defective nuclear mechanics and impaired mechanically activated gene transcription. These findings suggest that the tissue-specific effects of lamin A/C mutations observed in the laminopathies may arise from varying degrees of impaired nuclear mechanics and transcriptional activation.

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Year:  2004        PMID: 14755334      PMCID: PMC324542          DOI: 10.1172/JCI19670

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  39 in total

1.  Nuclear lamin A/C R482Q mutation in canadian kindreds with Dunnigan-type familial partial lipodystrophy.

Authors:  H Cao; R A Hegele
Journal:  Hum Mol Genet       Date:  2000-01-01       Impact factor: 6.150

2.  Contribution of the nucleus to the mechanical properties of endothelial cells.

Authors:  Nathalie Caille; Olivier Thoumine; Yanik Tardy; Jean-Jacques Meister
Journal:  J Biomech       Date:  2002-02       Impact factor: 2.712

3.  Lamin a truncation in Hutchinson-Gilford progeria.

Authors:  Annachiara De Sandre-Giovannoli; Rafaëlle Bernard; Pierre Cau; Claire Navarro; Jeanne Amiel; Irène Boccaccio; Stanislas Lyonnet; Colin L Stewart; Arnold Munnich; Martine Le Merrer; Nicolas Lévy
Journal:  Science       Date:  2003-04-17       Impact factor: 47.728

4.  LMNA, encoding lamin A/C, is mutated in partial lipodystrophy.

Authors:  S Shackleton; D J Lloyd; S N Jackson; R Evans; M F Niermeijer; B M Singh; H Schmidt; G Brabant; S Kumar; P N Durrington; S Gregory; S O'Rahilly; R C Trembath
Journal:  Nat Genet       Date:  2000-02       Impact factor: 38.330

5.  Missense mutations in the rod domain of the lamin A/C gene as causes of dilated cardiomyopathy and conduction-system disease.

Authors:  D Fatkin; C MacRae; T Sasaki; M R Wolff; M Porcu; M Frenneaux; J Atherton; H J Vidaillet; S Spudich; U De Girolami; J G Seidman; C Seidman; F Muntoni; G Müehle; W Johnson; B McDonough
Journal:  N Engl J Med       Date:  1999-12-02       Impact factor: 91.245

6.  MAPK and SRC-kinases control EGR-1 and NF-kappa B inductions by changes in mechanical environment in osteoblasts.

Authors:  C Granet; N Boutahar; L Vico; C Alexandre; M H Lafage-Proust
Journal:  Biochem Biophys Res Commun       Date:  2001-06-15       Impact factor: 3.575

7.  Essential roles for Caenorhabditis elegans lamin gene in nuclear organization, cell cycle progression, and spatial organization of nuclear pore complexes.

Authors:  J Liu; T Rolef Ben-Shahar; D Riemer; M Treinin; P Spann; K Weber; A Fire; Y Gruenbaum
Journal:  Mol Biol Cell       Date:  2000-11       Impact factor: 4.138

8.  Properties of lamin A mutants found in Emery-Dreifuss muscular dystrophy, cardiomyopathy and Dunnigan-type partial lipodystrophy.

Authors:  C Ostlund; G Bonne; K Schwartz; H J Worman
Journal:  J Cell Sci       Date:  2001-12       Impact factor: 5.285

9.  Loss of A-type lamin expression compromises nuclear envelope integrity leading to muscular dystrophy.

Authors:  T Sullivan; D Escalante-Alcalde; H Bhatt; M Anver; N Bhat; K Nagashima; C L Stewart; B Burke
Journal:  J Cell Biol       Date:  1999-11-29       Impact factor: 10.539

10.  Nuclear lamins A and B1: different pathways of assembly during nuclear envelope formation in living cells.

Authors:  R D Moir; M Yoon; S Khuon; R D Goldman
Journal:  J Cell Biol       Date:  2000-12-11       Impact factor: 10.539

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  421 in total

1.  LINCing lamin B2 to neuronal migration: growing evidence for cell-specific roles of B-type lamins.

Authors:  Catherine Coffinier; Loren G Fong; Stephen G Young
Journal:  Nucleus       Date:  2010 Sep-Oct       Impact factor: 4.197

Review 2.  How do mutations in lamins A and C cause disease?

Authors:  Howard J Worman; Jean-Claude Courvalin
Journal:  J Clin Invest       Date:  2004-02       Impact factor: 14.808

3.  The liquid drop nature of nucleoli.

Authors:  John F Marko
Journal:  Nucleus       Date:  2012-03-01       Impact factor: 4.197

4.  Biophysical assays to probe the mechanical properties of the interphase cell nucleus: substrate strain application and microneedle manipulation.

Authors:  Maria L Lombardi; Monika Zwerger; Jan Lammerding
Journal:  J Vis Exp       Date:  2011-09-14       Impact factor: 1.355

5.  Magnetic manipulation of nanorods in the nucleus of living cells.

Authors:  Alfredo Celedon; Christopher M Hale; Denis Wirtz
Journal:  Biophys J       Date:  2011-10-19       Impact factor: 4.033

Review 6.  The nuclear envelope as a chromatin organizer.

Authors:  Nikolaj Zuleger; Michael I Robson; Eric C Schirmer
Journal:  Nucleus       Date:  2011-09-01       Impact factor: 4.197

Review 7.  The nucleoskeleton as a genome-associated dynamic 'network of networks'.

Authors:  Dan N Simon; Katherine L Wilson
Journal:  Nat Rev Mol Cell Biol       Date:  2011-10-05       Impact factor: 94.444

Review 8.  Lamins at a glance.

Authors:  Chin Yee Ho; Jan Lammerding
Journal:  J Cell Sci       Date:  2012-05-01       Impact factor: 5.285

9.  How protein materials balance strength, robustness, and adaptability.

Authors:  Markus J Buehler; Yu Ching Yung
Journal:  HFSP J       Date:  2010-01-14

Review 10.  Spectrin and its interacting partners in nuclear structure and function.

Authors:  Muriel W Lambert
Journal:  Exp Biol Med (Maywood)       Date:  2018-03
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